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CASE REPORT Open Access The recurrence of colonic volvulus due to nonrotation after intestinal resection in adulthood: a case report Yusuke Sakimura 1* , Hirotaka Kitamura 1 , Noriyuki Inaki 2 and Hiroyuki Bando 1 Abstract Background: Intestinal nonrotation is a rare congenital condition that causes fatal colonic volvulus at any age. Once volvulus attack occurs, radical surgical therapy is required for treatment and the prevention of recurrence. This report describes the case of an adult female patient with a recurrence of cecum volvulus due to intestinal nonrotation after transverse colon resection for colonic volvulus. Case presentation: A 27-year-old female visited our emergency room (ER) with intermittent abdominal pain and nausea. Enhanced computed tomography (CT) showed enlargement of the level of the ascending and transverse colon and an obstruction with a whirlpool sign at the transverse colon. The small intestine was distributed on the right side of the abdominal cavity, and the large intestine occupied the left side. She was diagnosed with volvulus with intestinal nonrotation, and emergency surgery was performed. Surgical examination indicated that the ascending colon to the transverse colon was not fixed to the retroperitoneum, and the transverse colon was rotated 180° clockwise. The axis of the volvulus was a mesenteric adhesion of the transverse colon. The involved transverse colon was resected, and the intestine was reconstructed by functional end-to-end anastomosis (FEEA). Six years after the initial surgery, the patient presented to the ER with abdominal fullness and lower abdominal pain. Enhanced CT revealed that the cecum, ascending colon, and remaining transverse colon were dilated with an obstruction. The appendix was located in the left upper abdominal cavity. The clinical diagnosis was cecal volvulus with intestinal nonrotation. An emergency laparotomy revealed that the cecum was rotated 180° clockwise. The terminal ileum to the remaining transverse colon was resected, and FEEA was performed. Seven months later, she suffered obstruction of the intestine caused by an operative adhesion, and conservative treatment was successful. The patient has had no abdominal symptoms for one and a half years so far. Conclusions: Surgeons should realize that nonrotation of the intestines induces volvulus in adulthood and should familiarize themselves with its clinical findings, appropriate treatment, and prognosis. Even after surgical treatment, awareness of the recurrence of volvulus should be maintained to avoid a late diagnosis. Keywords: Intestinal volvulus, Intestinal rotational disorder, Nonrotation, Emergency surgery Background Colonic volvulus can cause bowel obstruction and lead to is- chemia, gangrene, and perforation of the involved segment, resulting in death [1]. Various diseases and conditions can induce volvulus of the large intestine, such as pregnancy, previous pelvic surgery, neuropsychological impairment, dia- betes, chronic constipation, and institutionalization [13]. Anomalies of intestinal rotation during the fetal period are crucial factors for the presentation of volvulus at any age [46]. The rate of adult patients varies among reports. Forty-eight percent of patients with this malrotation are diagnosed at an age over 18, either symptomatically or asymptomatically [7], but another study mentioned that approximately 75% of patients are diagnosed before 5 years of age [8]. The anomaly can provoke colonic volvulus in any segment of the large intestine at adulthood, and emergency surgery is needed. However, the prevalence and favored site of the colonic volvulus in adulthood is unclear, with limited © The Author(s). 2019 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. * Correspondence: [email protected] 1 Department of Gastroenterological Surgery, Ishikawa Prefectural Central Hospital, 2-1 Kuratuki Higashi, Kanazawa, Ishikawa 9208530, Japan Full list of author information is available at the end of the article Sakimura et al. Surgical Case Reports (2019) 5:147 https://doi.org/10.1186/s40792-019-0710-x

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Page 1: The recurrence of colonic volvulus due to nonrotation ......The clinical diagnosis was cecal volvulus with intestinal nonrotation. An emergency laparotomy revealed that the cecum was

CASE REPORT Open Access

The recurrence of colonic volvulus due tononrotation after intestinal resection inadulthood: a case reportYusuke Sakimura1* , Hirotaka Kitamura1, Noriyuki Inaki2 and Hiroyuki Bando1

Abstract

Background: Intestinal nonrotation is a rare congenital condition that causes fatal colonic volvulus at any age.Once volvulus attack occurs, radical surgical therapy is required for treatment and the prevention of recurrence. Thisreport describes the case of an adult female patient with a recurrence of cecum volvulus due to intestinalnonrotation after transverse colon resection for colonic volvulus.

Case presentation: A 27-year-old female visited our emergency room (ER) with intermittent abdominal pain andnausea. Enhanced computed tomography (CT) showed enlargement of the level of the ascending and transversecolon and an obstruction with a whirlpool sign at the transverse colon. The small intestine was distributed on theright side of the abdominal cavity, and the large intestine occupied the left side. She was diagnosed with volvuluswith intestinal nonrotation, and emergency surgery was performed. Surgical examination indicated that theascending colon to the transverse colon was not fixed to the retroperitoneum, and the transverse colon wasrotated 180° clockwise. The axis of the volvulus was a mesenteric adhesion of the transverse colon. The involvedtransverse colon was resected, and the intestine was reconstructed by functional end-to-end anastomosis (FEEA).Six years after the initial surgery, the patient presented to the ER with abdominal fullness and lower abdominalpain. Enhanced CT revealed that the cecum, ascending colon, and remaining transverse colon were dilated with anobstruction. The appendix was located in the left upper abdominal cavity. The clinical diagnosis was cecal volvuluswith intestinal nonrotation. An emergency laparotomy revealed that the cecum was rotated 180° clockwise. Theterminal ileum to the remaining transverse colon was resected, and FEEA was performed. Seven months later, shesuffered obstruction of the intestine caused by an operative adhesion, and conservative treatment was successful.The patient has had no abdominal symptoms for one and a half years so far.

Conclusions: Surgeons should realize that nonrotation of the intestines induces volvulus in adulthood and shouldfamiliarize themselves with its clinical findings, appropriate treatment, and prognosis. Even after surgical treatment,awareness of the recurrence of volvulus should be maintained to avoid a late diagnosis.

Keywords: Intestinal volvulus, Intestinal rotational disorder, Nonrotation, Emergency surgery

BackgroundColonic volvulus can cause bowel obstruction and lead to is-chemia, gangrene, and perforation of the involved segment,resulting in death [1]. Various diseases and conditions caninduce volvulus of the large intestine, such as pregnancy,previous pelvic surgery, neuropsychological impairment, dia-betes, chronic constipation, and institutionalization [1–3].

Anomalies of intestinal rotation during the fetal period arecrucial factors for the presentation of volvulus at any age[4–6]. The rate of adult patients varies among reports.Forty-eight percent of patients with this malrotation arediagnosed at an age over 18, either symptomatically orasymptomatically [7], but another study mentioned thatapproximately 75% of patients are diagnosed before 5 yearsof age [8]. The anomaly can provoke colonic volvulus in anysegment of the large intestine at adulthood, and emergencysurgery is needed. However, the prevalence and favored siteof the colonic volvulus in adulthood is unclear, with limited

© The Author(s). 2019 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, andreproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link tothe Creative Commons license, and indicate if changes were made.

* Correspondence: [email protected] of Gastroenterological Surgery, Ishikawa Prefectural CentralHospital, 2-1 Kuratuki Higashi, Kanazawa, Ishikawa 9208530, JapanFull list of author information is available at the end of the article

Sakimura et al. Surgical Case Reports (2019) 5:147 https://doi.org/10.1186/s40792-019-0710-x

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case reports [7, 9–14], and no case of recurrence at differentsegments has been reported. Every surgeon should knowthat an anomaly of the intestinal rotation is not only apediatric disease but that adult patients can also be affected[15]. Here, we report a rare case of recurrence of cecal vol-vulus after resection of the transverse colon for volvulus in afemale patient with undiagnosed nonrotation.

Case presentationFirst surgeryA 27-year-old female visited our emergency room (ER)with intermittent abdominal pain and nausea. Her lastbowel movement was 2 days before visiting. She had nonotable past medical history, including no abdominaloperation or growth abnormalities. On her physicalexamination, she was tachycardic, with a pulse of136 bpm, but the rest of her vital signs were normal. Ab-dominal examination revealed distension and epigastrictenderness with peritoneal irritation symptoms. Herlaboratory examination results showed slightly elevatedC-reactive protein levels (1.8 mg/dL), but the other find-ings were within normal ranges. Enhanced computedtomography (CT) showed enlargement at the level of theascending and transverse colon (Fig. 1). The transverse

colon had an obstructive point with a whirlpool sign.The colonic wall showed no ischemic signs, but the sup-ply vein was dilated. The large intestine was located onthe left side of the abdominal cavity, and the small intes-tine occupied the other side. The duodenum did notcross between the superior mesenteric artery (SMA) andthe abdominal aorta but went down straight on the rightside of the SMA. The SMA and the superior mesentericvein (SMV) were located at inverted positions; the SMAwas on the right side, and the SMV was on the left. Thepatient was diagnosed with transverse colonic volvulus,and nonrotation of the intestine was suspected.A transanal ileus tube was placed endoscopically at the

dilated colon to decompress the enlargement. The mu-cosa of the colon was congestive, but there were no nec-rotic findings (Fig. 2). She was treated conservatively andobserved for the next 24 h. However, her symptomsremained, and CT revealed no improvement in theintestinal obstruction and showed increasing ascites.Conservative therapy was determined to be ineffective;therefore, emergency surgery was performed. Thelaparoscopic view revealed the enlarged transverse colon,and there was no space for the surgical procedure. Con-sequently, the operation was converted to laparotomy

Fig. 1 Enhanced computed tomography before the first surgery. Transverse section (a) and coronary section (b) images show the enlarged transversecolon (T) with an obstructed point at the splenic flexure with a whirlpool sign (yellow arrow). The ascending colon (A), transverse colon (T), anddescending colon (D) were located on the left side of the abdominal cavity, and the small intestine (SI) was located on the other side. The cecum (C)inhabited the pelvic cavity. The superior mesenteric artery (red arrow) was located on the right side of the superior mesenteric vein (blue arrow). Theduodenum did not cross the vertebral column but traveled down straight (red circle). The drainage veins were dilated, but there were no signs ofischemia. c and d illustrate the gastrointestinal tract of nonrotation and volvulus at the transverse colon before surgery

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with a middle abdominal incision. The dilated transversecolon was pulled out from the abdominal cavity withoutany mobilization, and it was rotated with 180° clockwisealong a bridging adhesion within the transverse colon it-self. The dilated transverse colon was resected (Fig. 3)and reconstructed with functional end-to-end anasto-mosis (FEEA). The surgical observation also showed thatthe mesenteries of the ascending colon to the transversecolon had mobility without fixation to the retroperito-neum. There was no ligament formation requiringLadd’s procedure. The surgical diagnosis was transversecolonic volvulus, nonrotation type, due to the anomalyof bowel rotation. She was discharged uneventfully 12days after the surgery.

Second surgerySix years after the first surgery, the patient present tothe ER with abdominal fullness and lower abdominalpain. On physical examination, her vital signs were nor-mal. Abdominal examination revealed distension andlight lower abdominal tenderness without peritoneal irri-tation. Her laboratory data were within the normalrange. Enhanced CT revealed the dilated colon with anobstruction at the ascending colon (Fig. 4). Although a

focal beak sign and narrowing of the vein were noted,there was no sign of ischemia. The appendix was locatedon the left upper side of the abdominal cavity, unlike inthe previous surgery. Clinical diagnosis was volvulus ofthe cecum and intestinal nonrotation, and emergencylaparotomy was performed. In the surgery, the tractfrom the dilated cecum to the remaining transversecolon was extracted from the abdominal cavity withoutany resistance (Fig. 5). There was torsion of the terminalileum to the remaining transverse colon with a 180°clockwise rotation. The dilated and twisted tract was re-moved, and ileocolic anastomosis with FEEA was per-formed. The anastomotic site of the previous surgerywas adhered and fixed to surrounding tissue. She wasdischarged uneventfully on postoperative day 9. Al-though the patient was diagnosed with obstruction ofthe intestine caused by an operative adhesion 7 monthsafter the surgery, conservative treatment relieved hersymptoms in 3 days of hospitalization.

DiscussionThe true incidence of intestinal rotational disorders isunknown, although a report indicates that the ratio isapproximately 0.2–1% of the population and that

Fig. 2 Endoscopic findings before the first surgery. There was an obstructed area of the transverse colon (a), and the scope managed to passthrough the lesion. The oral side of the colon was dilated and contained with feces. The wall was conjugated and swollen, but no ischemicchange was observed (b)

Fig. 3 Pathological findings of the first surgery. The enlarged transverse colon was resected (a) and showed edema macroscopically. Pathologicalfindings (b) revealed edema and conjugated submucosal layers, and the mucosal layer showed hemorrhage and detachment. These findingswere inconsistent with the acute circulating disturbance

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patients present symptomatically at a rate of 1 in 2500[16]. This condition is the result of an error during theembryonic period. The classification of these anomaliesis divided into nonrotation, malrotation, incomplete ro-tation, paraduodenal hernia, and reverse rotation [5].Nonrotation is characterized by the duodenum travelingdescending straight down to the right side of the SMAand the small intestine occupying the right side of theabdominal cavity and the large intestine located on theleft side. Incomplete rotation or malrotation causes duo-denal obstruction due to the formation of Ladd’s bandsand the lack of duodenal loop rotation. This conditioncan lead to catastrophic midgut volvulus. Paraduodenalhernia is caused by failure of the 180° counterclockwise

rotation of the midgut. The small intestine herniates be-tween the ascending mesocolon and the retroperito-neum. Reversed rotation presents as the transverse colonlocated inferior to the duodenum and causes partialmesenteric arterial, venous, and lymphatic obstruction.Abnormal rotation of the intestine tends to be

discussed as a pediatric disease; however, adult patientssuffer from this condition with either acute or chronicsymptoms [15]. Acute symptoms are caused by suddenobstruction and ischemic changes in the intestine, as inour case. Chronic symptoms are not specific, such as ab-dominal pain, vomiting, and diarrhea, and can last sev-eral years [4, 7, 17, 18]. The diagnosis of nonrotation inadults is not easy before the appearance of acute

Fig. 4 Enhanced computed tomography before the second surgery. Transverse section (a) and coronary section (b) images show the enlargedcecum (C) and ascending colon (A). The obstructive point was on the remaining transverse colon with a whirlpool sign (red circle). Theanastomotic site of the first surgery (as) was placed in the left upper abdominal cavity. The appendix (white arrow) was in the upper abdominalcavity. The volvulus and the distribution of the gastrointestinal tract are indicated in (c)

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symptoms of obstruction. This is because patients usu-ally develop without any symptoms or with mild chronicsymptoms, and the number of cases is too small to iden-tify [7, 19]. Diagnosis is mainly conducted with CT scan,upper gastrointestinal examination, or incidental surgicalfindings [5]. However, radiographic examination is lim-ited to diagnosing the anomaly of the rotation due tofalse positives and negatives [17, 20, 21]. Operative find-ings are vital for the final diagnoses of an abnormality.In our case, the patient showed acute symptoms, and theCT findings suggested the possibility of volvulus andnonrotation [17, 22, 23]. Finally, the operative findingsconfirmed that she had nonrotation with volvulus.Colonic volvulus is the condition of bowel torsion

around its own mesentery and is the third leading causeof large intestine obstruction [2]. Intestinal volvulus pa-tients tend to have a long redundant colonic segmentand elongated mesentery with a narrow base [24, 25].These anatomical characteristics are either congenital oracquired. One of the congenital causes is the anomaly ofintestinal rotation [20, 26, 27]. This anomaly presents asa nonfixed colon and narrowed stalk formation withLadd’s bands or mesentery adhesions. In our opinion, incases of adult nonrotation patients, very loose volvulusintermittently occurs with light abdominal symptomssuch as pain and vomiting. Loose volvulus and highlymobilized mesentery may cause friction and inflamma-tion at the mesentery. Inflammation leads to the forma-tion of fibrous adhesions at the mesentery and narrowsthe stalk. Adhesions play a role to serve as an axis inacute severe volvulus at any age. Consequently, once thevolvulus attack due to a congenital intestinal rotationanomaly occurs, surgical resection of the involved intes-tine is the primary indication for radical surgery for

treatment and prevention of recurrence. The surgicalprocedure should be noted along with Ladd’s procedure[28, 29]. First, reduction of the volvulus is required. Sec-ond, any fixed band between the cecum or ascendingcolon and abdominal wall or the duodenum should be dis-sected to widen the stalk of the mesentery. Third, the ad-hesion around the duodenum should be detached tomobilize the proximal jejunum to the right upper quad-rant. Forth, the involved segment should be removed, andthe anastomotic site should be located far from the duode-num to avoid shortening the mesentery stalk, which maylead to further volvulus. Finally, the bowel should beplaced in a nonrotation position with appendectomy.There are several reports that indicate a second volvulusattack after Ladd’s procedure in infants for midgut volvu-lus [30]. This supports our rare case in which an appropri-ate surgical procedure was performed for volvulus withnonrotation, but volvulus recurrence occurred. Hence,what gastrointestinal surgeons should know is that the in-testinal rotation anomaly can suddenly affect adult pa-tients and lead to fatal volvulus, and volvulus recurrencemay occur even several years after surgery.

ConclusionsVolvulus in adulthood due to an anomaly of intestinalrotation is rare and requires surgical treatment. Al-though a relevant surgical procedure is performed, pa-tients can suffer recurrence as in our case. Hence,surgeons should recognize this condition as not only apediatric disease but also adult gastrointestinal disorder.

AbbreviationsCT: Computed tomography; ER: Emergency room; FEEA: Functional end-to-end anastomosis; SMA: Superior mesenteric artery; SMV: Superior mesentericvein

Fig. 5 Operative and pathological findings of the second surgery. The operative findings (a) showed that the cecum (C) and the appendix (blackarrow) were in the upper abdominal cavity, and the ascending colon ran from the cranial to the caudal side. The colon was pulled out from theabdominal cavity without any mobilization. The diagnosis was cecum volvulus with 180° rotation, and the tract was resected from the terminalileum to the remaining transverse colon (b). Pathological findings (c) revealed a thinned intestinal wall and edema at the mucosal andsubmucosal layers, which indicated an acute circulating disturbance

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AcknowledgementsThe authors would like to thank Springer Nature Author Services for Englishlanguage editing.

Authors’ contributionsYS drafted the manuscript and collected the references. HK supervised thepreparation of this report. YS and HB performed the second surgery. NIperformed the first surgery. All authors read and approved the final manuscript.

FundingThe authors declare that they received no funding support for this report.

Availability of data and materialsData sharing is not applicable to this article, because no datasets weregenerated or analyzed during this study.

Ethics approval and consent to participateNot applicable.

Consent for publicationWritten informed consent was obtained from the patient for the publicationof this case report, and the identity of the patient was protected.

Competing interestsThe authors declare that they have no competing interests.

Author details1Department of Gastroenterological Surgery, Ishikawa Prefectural CentralHospital, 2-1 Kuratuki Higashi, Kanazawa, Ishikawa 9208530, Japan.2Department of Surgery, Juntendo Urayasu Hospital, Juntendo University,2-1-1, Tomioka, Urayasu-shi, Chiba 2790021, Japan.

Received: 2 June 2019 Accepted: 27 September 2019

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